Thorax (1955), 10, 131.

MALIGNANT CHANGE IN BRONCHIECTASISBY

R. J. R. CURETON AND I. M. HILL

From St. Bartholomew's Hospital, London

(RECEIVED FOR PUBLICATION DECEMBER 10, 1954)

It has been known for some time (Deelman,1924; MacKenzie and Rous, 1941; Pullinger, 1943and 1945; Burrows and Clarkson, 1943; Burrows,Mayneord, and Roberts, 1937; Lacassagne, 1933;and others) that various types of injury can influencethe development of experimentally producedtumours, hastening their development and localizingtheir site. In man, many instances have beenrecorded where a cancer has been found closelyrelated to an area involved by former injury. Thus,for example, cancers have apparently developed inold burn scars (Treves and Pack, 1930), in associa-tion with varicose ulcers (Black, 1952), and some-times in relation to chronic inflammatory lesions(lupus vulgaris, syphilitic lesions, etc.). It is possiblethat cancer may have some aetiological relationwith such injury or chronic disease.

In the lung, instances have been recorded wherecancer has been found, apparently originating in thewalls of tuberculous cavities, in the " fibrous apicalcap" (James and Pagel, 1944) and, according toseveral authors including Schwartz (1950), inbronchial scars resulting from tuberculous ulcera-tion.

In recent years there have been recorded byseveral observers (Stewart and Allison, 1943;Petersen, Hunter, and Sneeden, 1949; Prior andJones, 1952; Raeburn and Spencer, 1953; andothers) a number of small early peripheral lungcancers, and evidence has been found that themajority of these tumours arose in areas of scarredlung.Among these tumours there have been several

which showed histological similarity to each otherand resembled, in many respects, the " carcinoid "type of bronchial adenoma and also the basal-celltumours of skin. The literature regarding theseneoplasms has been well reviewed in the articles byPrior and Jones and by Raeburn and Spencer.The case to be described falls into this group of

tumours, although in this instance the neoplastictissue was found widely disseminated in the lung.

CASE HISTORYW. H., a male commercial traveller aged 36 years, had

been investigated for cough and sputum of nine years'duration. At the age of 3 years he had had a lung infectionand an empyema on the left was drained for some time.The detailed history of this illness is unobtainable, butthree scars of drainage are present on the left chest wall.After this he remained well until the age of 27, when hedeveloped cough and sputum for the first time and wasinvalided from the Army.

His general condition remained good, but purulentsputum persisted, about Ij oz. being raised daily, mostlyat mid-day. In 1945 he had a haemoptysis of about 2 oz.on one occasion followed by occasional staining of thesputum.He was admitted to the Thoracic Department, St.

Bartholomew's Hospital, on April 24, 1952, for investi-gation, when he was found to have grade 3 clubbing ofthe fingers, markedly impaired movement and air entryin the left chest, moist sounds throughout the left lungfield, though mainly at the base, and some bronchospasmon both sides. Bronchoscopy on May 2 showed slightnarrowing of the left upper lobe orifice, but there was noother abnormality apart from thick pus in the left mainbronchus. Bilateral bronchograms showed totalbronchiectasis of all segments of the contracted leftlung, and a small area of cystic bronchiectasis in theright posterior and medial basal segments. Afterpreliminary chemotherapy to reduce the sputum as faras possible, a left pneumonectomy was carried out onJuly 25. Over the lower lobe, during the extrapleuralseparation of the lung, an epithelialized cavity wasopened at the site of the old rib resection and a bonesequestrum found within. Muscle flaps were used inclosing the chest wall, where excision of the scars hadleft a very thin chest wall.

Infection of the pneumonectomy space with penicillin-resistant microaerophilic streptococci followed. Thespace was sterilized with chloramphenicol solutioninjections and streptokinase, and a small wound sinusexcised and sutured. He was discharged healed andwell on September 20.

In the next year he remained well and almost sputumfree, but was readmitted to hospital on September 8,1953, with a fever which had failed to respond toaureomycin. Aspiration of the left pleural space revealed

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R. J. R. CURETON and LIL M. HILL

FIG. 1.-Slices of the shrunken, bronchiectatic left lung.

bloodstained pus, sterile aerobically, but growing amicroaerophilic streptococcus which was penicillinsensitive. The space was sterilized with daily injections of2m. units penicillin and he was discharged on October 2,only to be readmitted three weeks later, again with fever.This time the left pleural space was opened by ribresection, all the pus and clot evacuated under penicillincover, and the chest closed. The wound healed by primaryintention and he has remained well and afebrile since.At no time has there been any evidence of bronchialfistula and at the second thoracotomy on October 30there was no sign of recurrence of the tumour.

PATHOLOGYThe pneumonectomy specimen (Fig. 1) showed

the whole lower lobe and most of the upper lobeto be affected by an advanced degree of tubularbronchiectasis. The dilated bronchi were filled withgreyish mucus and little intervening lung tissuecould be identified in the lower lobe. In the upperlobe some aerated lung was found, although thiswas partly collapsed and haemorrhagic. The hilarlymph nodes were small and not obviously involvedby old or recent tuberculosis. Radiographs of thesliced specimen revealed no calcifications in thelymph nodes.

HISTOLOGYSections of both upper and lower lobes show a

widespread fibrosis of the lung tissue between theclosely crowded bronchiectatic structures. A gooddeal of adipose tissue is also found between areas of

scarring. The bronchi are extremely rugose due tofolding of the mucosa over very prominent musclebands. There is much proliferation of lymphoidtissue and hyperplasia of mucous glands in thebronchial walls (Figs. 2 and 3). Many bronchicontain pus. In numerous areas the muscle bandsare extremely prominent and often appear to becomemixed in a haphazard manner with the fibrous andadipose tissue. The irregular folding of the bronchialmucosa seems also to merge with " duct-like "extensions through the hypertrophied muscle coatof the bronchi (Fig. 4), and these extensions appearcontinuous with alveoli or other air spaces whichremain in the fibrotic areas and which are linedby a cubical epithelium (Fig. 5). Many of thesestructures show focal hyperplastic thickening (Figs.4, 6, and 7) and one can almost imagine a series ofgradations between this appearance and islands offrankly neoplastic tissue. The clumps of tumourcells (Figs. 8 and 9) have a certain regularity in theirappearance and have a distinct resemblance to thebasal-cell cancers of the skin.

In spite of their cytological appearances, however,the neoplastic masses are clearly infiltrating and,although perhaps not all of the spaces around thetumour clumps represent lymphatics, we thinknevertheless that some are indeed lymphatic channelslined by flattened endothelial cells (Fig. 9).Metastatic tumour masses are found also in thebronchopulmonary nodes and one tracheo-bronchiallymph node (Fig. 10).

132

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MALIGNANT CHANGE IN BRONCHIECTASIS

FIG. 2.-Folding of mucosa and hyper- N_plasia of lymphoid tissue andmucous glands with adjacent adi-pose tissue. (Haematoxylin andeosin, x 32.)

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,,; > <¢, <>. > w:F e>,& A.o3 e '; * e' & |5> * st SsiE $ Ri? ivr < .::<9... 5.^: " ..:- .¢.¢4's !ie !!,- ?WoV ..... iF - . : .. ' * . . - >.i . ig;S ?.is.'Mo> .. : ; 4; R ' ..... , 4, w°i,, ..... ,> F-F. 't m r3;S.l.5;;b . ::c:.Ms>> - : - :: -: -: .a:-^ .. : . . t,o. S8i M9fThe only other points of note are ossifying and

calcifying cartilages in the bronchi, prominentobliterative endarteritis, the absence of any activeor obsolete tuberculous lesions, and the fact that theneoplastic infiltration is found focally but widelydisseminated both in the upper and lower lobes,which leads us to believe that this tumour may havea multicentric origin.

DIscUSSIONThese peripheral lung tumours, resembling basal

cell cancer, appear to be in a small class of their

K

FIG. 3.-Folding of bronchial mucous

O membrane over prominent muscle

bands and clumps of neoplasticcells in fibrous tissue between thebronchi. (Haematoxylin and

M A........^eosin, x 14.)

own. They affect women more frequently than men,they have fairly distinctive histological features, andthey also appear to behave generally in a benignmanner. The previously recorded cases have,however, shown only small tumours in the peripherallung fields and these have been found either acciden-tally during examination of post-mortem materialor in pneumonectomy specimens. In each instancethe advance of the tumours has been effectivelyterminated. Thus, while some authors haveconsidered these neoplasms to represent oat-cell

133

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R. J. R. CURETON and L M. HILL

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FIG. 4.-The duct-like structuresextending through hypertrophiedmuscle bands. (Haematoxylinand eosin, x 92.)

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FIG. 5.-Air spaces remaining in thescar tissue and lined by cubicalepithelium. (Haematoxylin andeosin, x 116.)

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carcinomas, others have suggested that they are

benign growths. perhaps related to bronchial aden-omas. A multicentric origin has been suggestedin one of Raeburn and Spencer's cases and in a

case described by Gray and Cordonnier (1929).In 18 out of 24 recorded cases, the tumours have

been associated with bronchiectasis or bronchio-lectatic changes in the lung, and it seems likely thatthe neoplastic proliferation arises in connexionwith atypical cellular hyperplasia in areas of lungscarring. In spite of the frequency of bronchiectatic

changes in the lungs which have been observed inthese cases, it is clear that tumours are very

infrequently found in bronchiectasis. This rarityhas been noted by Tuttle and Womack (1934) andby Niskanen (1949), and in our series of bronchiec-tatic specimens at St. Bartholomew's Hospital thishas been the only neoplasm found in 54 cases. It ispossible, however, that some tumours may havebeen missed owing to their small size. Stewart andAllison (1943), for instance, found by accident, in a

histological section from a bronchiectatic lung, a

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MALIGNANT CHANGE IN BRONCHIECTASIS

Fios. 6 and 7.-Haematoxylin and eosin, x 155. Apparent transition between epithelial hyperplasia and neoplastic proliferation.

FIG. 8.-Clumps of neoplastic cells in scar tissue. (Haematoxylin and eosin, x 190.)

135

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FIG. 9.-Tumour clumps apparently infiltrating lymphatics. Theresemblance to basal-cell tumours is apparent. (Haematoxylinand eosin, x 100.)

microscopic focus of growth 1.0 mm. x 0.4 mm.which had cytological features identical with thosedescribed above. Even in the case described here,where the tumour was widespread in all three lobes,its presence was only recognized when the histo-logical sections were examined.Womack and Graham (1941) have suggested that

this type of tumour is associated with a congenitalabnormality of the lung. We have found no evidencein our case to support this.

It is possible that the subsequent history of thiscase may indicate how malignant and how rapidlygrowing these tumours may be, for, if the neoplasmis arising multicentrically in relation to thebronchiectatic changes, similar neoplastic tissuemay be present in the basal segments of the rightlung.

It is at least evident from the histological charac-teristics that the tumour in this case is locallymalignant with powers of metastasizing to theregional lymph nodes.

SUMMARYThe history and pathological appearances are

described in a case of lung tumour associated withbronchiectasis.

FIG. 10.-Tumour metastasis in a bronchopulmonary lymph node.(Haematoxylin and eosin, 160.)

The histological features, which resemble thebasal-cell carcinomas of skin, are compared withsimilar tumours described in the literature.The malignant nature of this particular tumour is

indicated.

We should like to thank Professor J. W. S. Blacklockfor much helpful criticism, Mr. J. W. Miller for histo-logical sections, Mr. N. K. Harrison for the photographof the lung specimen, and Dr. G. S. Sansom and Mr.K. W. Iles for the photomicrographs.

REFERENCESBlack, W. (1952). Brit. J. Cancer, 6, 120.Burrows, H., and Clarkson, J. R. (1943). Brit. J. Radiol., 16, 381.

Mayneord, W. V., and Roberts, J. E. (1937). Proc. roy. Soc.B., 123, 213.

Deelman, H. T. (1924). Z. Krebsforsch., 21, 220.Gray, S. H., and Cordonnier, J. (1929). Arch. Surg., Chicago, 19,

1618.James, I., and Pagel, W. (1944). Brit. J. Surg., 32, 85.Lacassagne, A. (1933). C.R. Soc. Biol., Paris, 112, 562.MacKenzie, I.. and Rous, P. (1941). J. exp. Med., 73, 391.Niskanen, K. 0. (1949). Acta path. microbiol. scand., Suppl. 80.Petersen, A. B., Hunter, W. C., and Sneeden, V. D. (1949). Cancer,

2, 991.Prior, J. T., and Jones, D. B. (1952). J. thorac. Surg., 23, 224.Pullinger, B. D. (1943). J. Path. Bact., 55, 301.-(1945). Ibid., 57, 467.Raeburn, C., and Spencer, H. (1953). Thorax, 8, 1.Schwartz, P. (1950). Beitr. Klin. Tuberk., 103, 192.Stewart, M. J., and Allison, P. R. (1943). J. Path. Bact., 55, 105.Treves, N., and Pack, G. T. (1930). Surg. Gynec. Obstet., 51, 749.Tuttle, W. M., and Womack, N. A. (1934). J. thorac. Surg., 4, 125.Womack, N. A., and Graham, E. A. (1941). Amer. J. Path.,17, 645.

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BronchiectasisMalignant Change in

R. J. R. Cureton and I. M. Hill

doi: 10.1136/thx.10.2.1311955 10: 131-136 Thorax 

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